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Candida epiglottitis in an adult with acute nonlymphocytic leukemia
Candida Epiglottitis in an Adult with Acute Nonlymphocytic Leukemia
SOPHIE COLE, B.S. MARLENE ZAWIN, M.D. BRUCE LUNDBERG, M.D. JOE HOFFMAN, M.D. LEE BAILEY, B.S. MARC S. ERNSTOFF, M.D.*
A 40-year-old white woman presented with fever, otalgia, and odynophagia and was found to have a peripheral white blood cell count of 90,000/mm3. A diagnosls of acute myekqenous leukemia was made. Further evaluation of symptoms and source for fever led to the diagnosis of Candida albicans epiglottitis. This is the first reported case of fungal epiglottitis in an immunocompromised adult.
New Haven, Connecticut
Epiglottitis occurs more frequently in adults than is generally realized [l-8]. The overwhelming majority of cases are caused by bacterial pathogens (gram-positive cocci, Hemophilus influenzae). Fungal infection as a cause of epiglottitis has only been reported in one patient [3] and is therefore unusual. One of the major medical complications of acute nonlymphocytic leukemia is infection with multiple pathogens including gram-negative bacteria, fungal and protozoa1 species, and intracellular bacteria. Epiglottitis is a rare entity in patients with acute nonlymphocytic leukemia, despite their immunocompromised state. We report an unusual case of opportunistic Candida epiglottitis in an adult with acute nonlymphocytic leukemia.
CASE REPORT
From the Yale Comprehensive Cancer Center, the Section of Medical Oncology, and the Departments of Medicine and Diagnostic Imaging, Yale University School of Medicine, New Haven, Connecticut. This work was supported in part by National Institutes of Health Grant qA-08341-20. Dr. Ernstoff is the recipient of an American Cancer Society Junior Faculty Award and a Pharmaceutical Manufacturers Foundation Association Award. Manuscript submitted February 27, 1986, and accepted March 27, 1986. *Current address and address for reprint requests: University of Pittsburgh School of Medicine and the Pittsburgh Cancer Institute, 230 Lothrop Street, Pittsburgh, Pennsylvania 15213.
662
March
23, 1967
The American
Journal
A 40-year-old housewife was in otherwise good health until three weeks prior to admission when she noted the onset of sore throat and dysphagia. She was initially treated with penicillin with no response. Otalgia, odynophagia, and fever of 101’F subsequently developed three days prior to admission. She was found then to have a white blood cell count of 90,000/mm3 with 90 percent blast forms. There was no history of hoarseness, drooling, stridor, or signs of upper airway obstruction. Physical examination upon admission to Yale-New Haven Hospital revealed a well-developed woman in no respiratory distress. The pulse was 100 beats/minute, blood pressure 120/80 mm Hg, temperature 99.4OF, and respiratory rate 20/minute. The oral cavity and oropharynx were mildly erythematous. She had mild, diffuse, tender lymphadenopathy of her neck. White blood cell count was 99,000/mm3 with 95 percent myeloblasts and with Auer rods present within the myeloblasts. Further hematologic work-up confirmed a diagnosis of acute myelogenous leukemia. Treatment with daunomycin (45 mg/m*), &thioguanine (160 mg/m*), and cytosine arabinoside (100 mg/hour) was begun. Because of fever, administration of broad-spectrum antibiotics was initiated. She continued to complain of sore throat and otalgia, and indirect otolaryngologic examination revealed an erythematous and swollen epiglottis with a cheesy exudate. The supraglottic area was also erythematous and swollen. The vocal cords and subglottic area were normal. A swab of the
epiglottis showed a moderate amount of budding yeasts and mycelia consistent with Candida albicans. Throat culture also revealed
of Medicine
Volume
62
EPIGLOTTITIS
IN A LEUKEMIC
PATIENT-COLE
ET AL
Figure 1. Left, initial lateral soft tissue film of the neck obtained on August 14, 1985, reveals thickening of the epiglottis (arrow). The subglottic area is normal. These findings are compatible with epip lottitis. Right, follow-up examination of August 20, 1985, reveals a normal ap pearance of the epiglottis with resolution of the prior edema (arrow).
C. albicans. Blood and sputum cultures showed no growth. Lateral neck radiography revealed a markedly swollen epiglottis without airway obstruction (Figure 1, left). Amphotericin B was administered intravenously (a total dose of 470 mg was given). Complete resolution of signs and symptoms and recovery were uneventful. Repeated lateral neck radiography showed resolution of swelling of the epiglottis (Figure 1, right).
Epiglottitis is well known to be a disease of bacterial cause. Branefors-Helander et al [l] reviewed 25 patients with epiglottitis; H. influenzae type B was isolated as the causative organism in 18 patients, one patient had a Diplococcus pneumoniae infection, and no pathogen was isolated in the remaining six patients. In another review, Hawkins et al [4] noted that the causative organism most often isolated in adults with acute epiglottitis was H. influenzae type B. They also noted that epiglottitis is more severe when due to H. influenzae type B than when due to other pathogens. The isolation of a fungal pathogen in epiglottitis is very rare. In a review of the literature, only one previous case of Candida epiglottitis in a healthy, nondebilitated patient has been reported [5]. However, the finding of oppottunistic fungal epiglottitis in an immunocompromised patient
March
has not been previously reported in the literature. The characteristics of the disease in the present case are unusual in that the patient had nonspecific symptoms of sore throat and otalgia. She did not have stridor or the usual acutely ill appearance that is expected in epiglottitis. In a review of 80 cases of adult epiglottitis, Deeb et al [2] found that none of the patients who presented more than 12 hours after the onset of symptoms had drooling or signs of upper airway obstruction, and all responded to medical treatment without airway intervention. This finding was explained by the fact that the inflammatory process had peaked before the patients sought medical help, and that the disease had already begun to resolve. Because of her immunocompromised state, our patient was not able to have a sufficient inflammatory reaction, and as a result, obstruction of the laryngeal airway did not develop. However, regardless of the immunologic status of the patient, epiglottitis in adults should be viewed as a lifethreatening disease, and strict precautionary measures should be undertaken to secure an adequate airway. Finally, it must be emphasized that in immunocompromised patients with persistent, nonspecific complaints of sore throat, odynophagia, and otalgia that cannot be explained by routine oropharyngeal inspection, further reevaluation with indirect laryngoscopy and lateral neck radiography should be carried out to rule out epiglottitis.
23, 1987
The American
Journal
of Medicine
Volume
82
663
EPIGLOTTITIS
IN A LEUKEMIC
PATIENT-COLE
ET AL
REFERENCES 1.
2. 3. 4.
664
Branefors-Helander P, Jeppsson P: Acute epiglottitis: a clinical, bacteriological and serological study. Stand J Infect Dis 1975; 7: 103-111. Deeb ZE, Yenson AC, DeFries HO: Acute epiglottitis in adults. Laryngoscope 1985; 95: 289-291. Haberman RS, Becker M, Ford C: Candida epiglottitis. Arch Otolaryngol 1983; 109: 770-771. Hawkins D, Miller AH, Sachs GB, Benz RT: Acute epiglottitis in adults. Laryngoscope 1973; 83: 1200-1211.
March
23, 1967
The American
Journal
of Medicine
Lowell SY: Nosocomial infections in the immunocompromised host. Am J Med 1981; 70: 398-404. Morgenstein JM, Abramson AL: Acute epiglottitis in adults. Laryngoscope 1971; 81: 1066-1073. Ossof RH, Wolff AP, Ballenger JJ: Acute epiglottitis in adults: experience with fifteen cases. Laryngoscope 1980; 90: 11551161. Robbins JP, FitzHugh GS: Epiglottitis in the adult. Laryngoscope 1971; 81: 700-706.
Volume
62
SOPHIE COLE, B.S. MARLENE ZAWIN, M.D. BRUCE LUNDBERG, M.D. JOE HOFFMAN, M.D. LEE BAILEY, B.S. MARC S. ERNSTOFF, M.D.*
A 40-year-old white woman presented with fever, otalgia, and odynophagia and was found to have a peripheral white blood cell count of 90,000/mm3. A diagnosls of acute myekqenous leukemia was made. Further evaluation of symptoms and source for fever led to the diagnosis of Candida albicans epiglottitis. This is the first reported case of fungal epiglottitis in an immunocompromised adult.
New Haven, Connecticut
Epiglottitis occurs more frequently in adults than is generally realized [l-8]. The overwhelming majority of cases are caused by bacterial pathogens (gram-positive cocci, Hemophilus influenzae). Fungal infection as a cause of epiglottitis has only been reported in one patient [3] and is therefore unusual. One of the major medical complications of acute nonlymphocytic leukemia is infection with multiple pathogens including gram-negative bacteria, fungal and protozoa1 species, and intracellular bacteria. Epiglottitis is a rare entity in patients with acute nonlymphocytic leukemia, despite their immunocompromised state. We report an unusual case of opportunistic Candida epiglottitis in an adult with acute nonlymphocytic leukemia.
CASE REPORT
From the Yale Comprehensive Cancer Center, the Section of Medical Oncology, and the Departments of Medicine and Diagnostic Imaging, Yale University School of Medicine, New Haven, Connecticut. This work was supported in part by National Institutes of Health Grant qA-08341-20. Dr. Ernstoff is the recipient of an American Cancer Society Junior Faculty Award and a Pharmaceutical Manufacturers Foundation Association Award. Manuscript submitted February 27, 1986, and accepted March 27, 1986. *Current address and address for reprint requests: University of Pittsburgh School of Medicine and the Pittsburgh Cancer Institute, 230 Lothrop Street, Pittsburgh, Pennsylvania 15213.
662
March
23, 1967
The American
Journal
A 40-year-old housewife was in otherwise good health until three weeks prior to admission when she noted the onset of sore throat and dysphagia. She was initially treated with penicillin with no response. Otalgia, odynophagia, and fever of 101’F subsequently developed three days prior to admission. She was found then to have a white blood cell count of 90,000/mm3 with 90 percent blast forms. There was no history of hoarseness, drooling, stridor, or signs of upper airway obstruction. Physical examination upon admission to Yale-New Haven Hospital revealed a well-developed woman in no respiratory distress. The pulse was 100 beats/minute, blood pressure 120/80 mm Hg, temperature 99.4OF, and respiratory rate 20/minute. The oral cavity and oropharynx were mildly erythematous. She had mild, diffuse, tender lymphadenopathy of her neck. White blood cell count was 99,000/mm3 with 95 percent myeloblasts and with Auer rods present within the myeloblasts. Further hematologic work-up confirmed a diagnosis of acute myelogenous leukemia. Treatment with daunomycin (45 mg/m*), &thioguanine (160 mg/m*), and cytosine arabinoside (100 mg/hour) was begun. Because of fever, administration of broad-spectrum antibiotics was initiated. She continued to complain of sore throat and otalgia, and indirect otolaryngologic examination revealed an erythematous and swollen epiglottis with a cheesy exudate. The supraglottic area was also erythematous and swollen. The vocal cords and subglottic area were normal. A swab of the
epiglottis showed a moderate amount of budding yeasts and mycelia consistent with Candida albicans. Throat culture also revealed
of Medicine
Volume
62
EPIGLOTTITIS
IN A LEUKEMIC
PATIENT-COLE
ET AL
Figure 1. Left, initial lateral soft tissue film of the neck obtained on August 14, 1985, reveals thickening of the epiglottis (arrow). The subglottic area is normal. These findings are compatible with epip lottitis. Right, follow-up examination of August 20, 1985, reveals a normal ap pearance of the epiglottis with resolution of the prior edema (arrow).
C. albicans. Blood and sputum cultures showed no growth. Lateral neck radiography revealed a markedly swollen epiglottis without airway obstruction (Figure 1, left). Amphotericin B was administered intravenously (a total dose of 470 mg was given). Complete resolution of signs and symptoms and recovery were uneventful. Repeated lateral neck radiography showed resolution of swelling of the epiglottis (Figure 1, right).
Epiglottitis is well known to be a disease of bacterial cause. Branefors-Helander et al [l] reviewed 25 patients with epiglottitis; H. influenzae type B was isolated as the causative organism in 18 patients, one patient had a Diplococcus pneumoniae infection, and no pathogen was isolated in the remaining six patients. In another review, Hawkins et al [4] noted that the causative organism most often isolated in adults with acute epiglottitis was H. influenzae type B. They also noted that epiglottitis is more severe when due to H. influenzae type B than when due to other pathogens. The isolation of a fungal pathogen in epiglottitis is very rare. In a review of the literature, only one previous case of Candida epiglottitis in a healthy, nondebilitated patient has been reported [5]. However, the finding of oppottunistic fungal epiglottitis in an immunocompromised patient
March
has not been previously reported in the literature. The characteristics of the disease in the present case are unusual in that the patient had nonspecific symptoms of sore throat and otalgia. She did not have stridor or the usual acutely ill appearance that is expected in epiglottitis. In a review of 80 cases of adult epiglottitis, Deeb et al [2] found that none of the patients who presented more than 12 hours after the onset of symptoms had drooling or signs of upper airway obstruction, and all responded to medical treatment without airway intervention. This finding was explained by the fact that the inflammatory process had peaked before the patients sought medical help, and that the disease had already begun to resolve. Because of her immunocompromised state, our patient was not able to have a sufficient inflammatory reaction, and as a result, obstruction of the laryngeal airway did not develop. However, regardless of the immunologic status of the patient, epiglottitis in adults should be viewed as a lifethreatening disease, and strict precautionary measures should be undertaken to secure an adequate airway. Finally, it must be emphasized that in immunocompromised patients with persistent, nonspecific complaints of sore throat, odynophagia, and otalgia that cannot be explained by routine oropharyngeal inspection, further reevaluation with indirect laryngoscopy and lateral neck radiography should be carried out to rule out epiglottitis.
23, 1987
The American
Journal
of Medicine
Volume
82
663
EPIGLOTTITIS
IN A LEUKEMIC
PATIENT-COLE
ET AL
REFERENCES 1.
2. 3. 4.
664
Branefors-Helander P, Jeppsson P: Acute epiglottitis: a clinical, bacteriological and serological study. Stand J Infect Dis 1975; 7: 103-111. Deeb ZE, Yenson AC, DeFries HO: Acute epiglottitis in adults. Laryngoscope 1985; 95: 289-291. Haberman RS, Becker M, Ford C: Candida epiglottitis. Arch Otolaryngol 1983; 109: 770-771. Hawkins D, Miller AH, Sachs GB, Benz RT: Acute epiglottitis in adults. Laryngoscope 1973; 83: 1200-1211.
March
23, 1967
The American
Journal
of Medicine
Lowell SY: Nosocomial infections in the immunocompromised host. Am J Med 1981; 70: 398-404. Morgenstein JM, Abramson AL: Acute epiglottitis in adults. Laryngoscope 1971; 81: 1066-1073. Ossof RH, Wolff AP, Ballenger JJ: Acute epiglottitis in adults: experience with fifteen cases. Laryngoscope 1980; 90: 11551161. Robbins JP, FitzHugh GS: Epiglottitis in the adult. Laryngoscope 1971; 81: 700-706.
Volume
62